Non ketotic hyperglycinemia is a rare inborn error of glycine metabolism due to deficient activity of glycine cleavage system, a multienzymatic complex consisting of four protein subunits: the P-protein, the H-protein, the T-protein and the L-protein. The neonatal form of non ketotic hyperglycinemia presents in the first days of life with encephalopathy, seizures, multifocal myoclonus and characteristic "hiccups". Rapid progression may lead to intractable seizures, coma and respiratory failure requiring mechanical ventilation. Clinical trial with scavenges drugs decreasing glycine levels such as sodium benzoate, and with drugs reducing NMDA receptors excitatory properties, such as ketamine and dextromethorphan, have been tried but the outcome is usually poor; antiepileptic therapy, moreover, is unable to control epileptic seizures. Ketogenic diet has been successfully tried for refractory epilepsy in pediatric patients. We report three cases affected by neonatal non ketotic hyperglycinemia and early myoclonic encephalopathy treated with ketogenic diet. In our patients ketogenic diet, in association with standard pharmacological therapy, determined dramatic reduction of seizures and improved quality of life.

Cusmai, R., Martinelli, D., Moavero, R., Dionisi Vici, C., Vigevano, F., Castana, C., et al. (2012). Ketogenic diet in early myoclonic encephalopathy due to non ketotic hyperglycinemia. EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY, 16(5), 509-513 [10.1016/j.ejpn.2011.12.015].

Ketogenic diet in early myoclonic encephalopathy due to non ketotic hyperglycinemia

MOAVERO, ROMINA;
2012-09-01

Abstract

Non ketotic hyperglycinemia is a rare inborn error of glycine metabolism due to deficient activity of glycine cleavage system, a multienzymatic complex consisting of four protein subunits: the P-protein, the H-protein, the T-protein and the L-protein. The neonatal form of non ketotic hyperglycinemia presents in the first days of life with encephalopathy, seizures, multifocal myoclonus and characteristic "hiccups". Rapid progression may lead to intractable seizures, coma and respiratory failure requiring mechanical ventilation. Clinical trial with scavenges drugs decreasing glycine levels such as sodium benzoate, and with drugs reducing NMDA receptors excitatory properties, such as ketamine and dextromethorphan, have been tried but the outcome is usually poor; antiepileptic therapy, moreover, is unable to control epileptic seizures. Ketogenic diet has been successfully tried for refractory epilepsy in pediatric patients. We report three cases affected by neonatal non ketotic hyperglycinemia and early myoclonic encephalopathy treated with ketogenic diet. In our patients ketogenic diet, in association with standard pharmacological therapy, determined dramatic reduction of seizures and improved quality of life.
set-2012
Pubblicato
Rilevanza internazionale
Articolo
Esperti anonimi
Settore MED/39 - NEUROPSICHIATRIA INFANTILE
English
Con Impact Factor ISI
Anticonvulsants; Combined Modality Therapy; Female; Humans; Hyperglycinemia, Nonketotic; Infant; Infant, Newborn; Male; Opsoclonus-Myoclonus Syndrome; Treatment Outcome; Ketogenic Diet
Cusmai, R., Martinelli, D., Moavero, R., Dionisi Vici, C., Vigevano, F., Castana, C., et al. (2012). Ketogenic diet in early myoclonic encephalopathy due to non ketotic hyperglycinemia. EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY, 16(5), 509-513 [10.1016/j.ejpn.2011.12.015].
Cusmai, R; Martinelli, D; Moavero, R; Dionisi Vici, C; Vigevano, F; Castana, C; Elia, M; Bernabei, S; Bevivino, E
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Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/2108/168161
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